Distinct phenotypes in mixed connective tissue disease: subgroups and survival
The aim of the present study was to assess the autoantibody profile, dominant clinical symptoms and cluster characteristics of different Mixed connective tissue disease (MCTD phenotypes. Two-hundred-and-one patients with MCTD were followed-up longitudinally. Five clinical parameters, Raynaud’s pheno...
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| Veröffentlicht in: | Lupus 2012-11, Vol.21 (13), p.1412-1422 |
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| creator | Szodoray, P Hajas, A Kardos, L Dezso, B Soos, G Zold, E Vegh, J Csipo, I Nakken, B Zeher, M Szegedi, G Bodolay, E |
| description | The aim of the present study was to assess the autoantibody profile, dominant clinical symptoms and cluster characteristics of different Mixed connective tissue disease (MCTD phenotypes. Two-hundred-and-one patients with MCTD were followed-up longitudinally. Five clinical parameters, Raynaud’s phenomenon, pulmonary artery hypertension (PAH), myositis, interstitial lung disease (ILD), erosive arthritis and five auto-antibodies besides anti-U1RNP, antiendothelial cell antibodies (AECA), anti-CCP, anti-cardiolipin (anti-CL), anti-SSA/SSB and IgM rheumatoid factor (RF) were selected for cluster analysis. The mean age of patients was 52.9 ± 12.4 years and the mean follow-up of the disease was 12.5 ± 7.2 years. Patients were classified into three cluster groups. Cluster 1 with 77 patients, cluster 2 with 79 patients and cluster 3 with 45 patients. In cluster 1 the prevalence of PAH (55.8%; p |
| doi_str_mv | 10.1177/0961203312456751 |
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Two-hundred-and-one patients with MCTD were followed-up longitudinally. Five clinical parameters, Raynaud’s phenomenon, pulmonary artery hypertension (PAH), myositis, interstitial lung disease (ILD), erosive arthritis and five auto-antibodies besides anti-U1RNP, antiendothelial cell antibodies (AECA), anti-CCP, anti-cardiolipin (anti-CL), anti-SSA/SSB and IgM rheumatoid factor (RF) were selected for cluster analysis. The mean age of patients was 52.9 ± 12.4 years and the mean follow-up of the disease was 12.5 ± 7.2 years. Patients were classified into three cluster groups. Cluster 1 with 77 patients, cluster 2 with 79 patients and cluster 3 with 45 patients. In cluster 1 the prevalence of PAH (55.8%; p < 0.001), Raynaud’s phenomenon (92.2%; p < 0.001) and livedo reticularis (24.6%, p < 0.001) was significantly greater than in cluster 2 and 3. In cluster 2, the incidence of ILD (98.7%; p < 0.001), myositis (77.2%; p < 0.001), and esophageal dysmotility (89.8%; p < 0.001) was significantly greater than that in cluster 1 and 3. In cluster 3, anti-CCP antibodies were present in 31 of 45 patients (68.8%) with erosions. Anti-CCP antibodies were present in 37 of 42 patients (88.0%) with erosions. PAH, angina, venous thrombosis was observed in cluster 1 and pulmonary fibrosis in cluster 2, musculosceletal damage, gastrointestinal symptoms and osteoporotic fractures were most frequent in cluster 3. Cumulative survival assessment indicated cluster 1 patients having the worst prognosis. Cluster analysis is valuable to differentiate among various subsets of MCTD and useful prognostic factor regarding the disease course.]]></description><identifier>ISSN: 0961-2033</identifier><identifier>EISSN: 1477-0962</identifier><identifier>DOI: 10.1177/0961203312456751</identifier><identifier>PMID: 22864236</identifier><language>eng</language><publisher>London, England: SAGE Publications</publisher><subject>Adult ; Age ; Aged ; Analysis of Variance ; Angina ; Antibodies ; Arthritis ; Arthritis - epidemiology ; Autoantibodies ; Autoantibodies - blood ; Biomarkers - blood ; Biopsy ; cardiolipin ; Chi-Square Distribution ; Cluster Analysis ; Connective tissue diseases ; Disease Progression ; Esophagus ; Familial Primary Pulmonary Hypertension ; Female ; Fibrosis ; Fractures ; Hospitals ; Humans ; Hungary - epidemiology ; Hypertension ; Hypertension, Pulmonary - epidemiology ; Immunoglobulin M ; Immunology ; Incidence ; Investigations ; Longitudinal Studies ; Lung diseases ; Lung Diseases, Interstitial - epidemiology ; Lupus ; Male ; Medical prognosis ; Middle Aged ; Mixed connective tissue disease ; Mixed Connective Tissue Disease - classification ; Mixed Connective Tissue Disease - diagnosis ; Mixed Connective Tissue Disease - epidemiology ; Mixed Connective Tissue Disease - immunology ; Mixed Connective Tissue Disease - mortality ; Myositis ; Myositis - epidemiology ; Osteoporosis ; Phenotype ; Prevalence ; Prognosis ; Pulmonary arteries ; Pulmonary artery ; Raynaud disease ; Raynaud Disease - epidemiology ; Rheumatoid factor ; Survival ; Survival Analysis ; Thrombosis ; Time Factors</subject><ispartof>Lupus, 2012-11, Vol.21 (13), p.1412-1422</ispartof><rights>The Author(s), 2012. Reprints and permissions: http://www.sagepub.co.uk/journalsPermissions.nav</rights><rights>SAGE Publications © Nov 2012</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c464t-8a0092599c38fe561d5d2d43b3dde91b8ab5e3e75799ce052a57bf04452d25133</citedby><cites>FETCH-LOGICAL-c464t-8a0092599c38fe561d5d2d43b3dde91b8ab5e3e75799ce052a57bf04452d25133</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://journals.sagepub.com/doi/pdf/10.1177/0961203312456751$$EPDF$$P50$$Gsage$$H</linktopdf><linktohtml>$$Uhttps://journals.sagepub.com/doi/10.1177/0961203312456751$$EHTML$$P50$$Gsage$$H</linktohtml><link.rule.ids>314,776,780,21799,27903,27904,43600,43601</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/22864236$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Szodoray, P</creatorcontrib><creatorcontrib>Hajas, A</creatorcontrib><creatorcontrib>Kardos, L</creatorcontrib><creatorcontrib>Dezso, B</creatorcontrib><creatorcontrib>Soos, G</creatorcontrib><creatorcontrib>Zold, E</creatorcontrib><creatorcontrib>Vegh, J</creatorcontrib><creatorcontrib>Csipo, I</creatorcontrib><creatorcontrib>Nakken, B</creatorcontrib><creatorcontrib>Zeher, M</creatorcontrib><creatorcontrib>Szegedi, G</creatorcontrib><creatorcontrib>Bodolay, E</creatorcontrib><title>Distinct phenotypes in mixed connective tissue disease: subgroups and survival</title><title>Lupus</title><addtitle>Lupus</addtitle><description><![CDATA[The aim of the present study was to assess the autoantibody profile, dominant clinical symptoms and cluster characteristics of different Mixed connective tissue disease (MCTD phenotypes. Two-hundred-and-one patients with MCTD were followed-up longitudinally. Five clinical parameters, Raynaud’s phenomenon, pulmonary artery hypertension (PAH), myositis, interstitial lung disease (ILD), erosive arthritis and five auto-antibodies besides anti-U1RNP, antiendothelial cell antibodies (AECA), anti-CCP, anti-cardiolipin (anti-CL), anti-SSA/SSB and IgM rheumatoid factor (RF) were selected for cluster analysis. The mean age of patients was 52.9 ± 12.4 years and the mean follow-up of the disease was 12.5 ± 7.2 years. Patients were classified into three cluster groups. Cluster 1 with 77 patients, cluster 2 with 79 patients and cluster 3 with 45 patients. In cluster 1 the prevalence of PAH (55.8%; p < 0.001), Raynaud’s phenomenon (92.2%; p < 0.001) and livedo reticularis (24.6%, p < 0.001) was significantly greater than in cluster 2 and 3. In cluster 2, the incidence of ILD (98.7%; p < 0.001), myositis (77.2%; p < 0.001), and esophageal dysmotility (89.8%; p < 0.001) was significantly greater than that in cluster 1 and 3. In cluster 3, anti-CCP antibodies were present in 31 of 45 patients (68.8%) with erosions. Anti-CCP antibodies were present in 37 of 42 patients (88.0%) with erosions. PAH, angina, venous thrombosis was observed in cluster 1 and pulmonary fibrosis in cluster 2, musculosceletal damage, gastrointestinal symptoms and osteoporotic fractures were most frequent in cluster 3. Cumulative survival assessment indicated cluster 1 patients having the worst prognosis. Cluster analysis is valuable to differentiate among various subsets of MCTD and useful prognostic factor regarding the disease course.]]></description><subject>Adult</subject><subject>Age</subject><subject>Aged</subject><subject>Analysis of Variance</subject><subject>Angina</subject><subject>Antibodies</subject><subject>Arthritis</subject><subject>Arthritis - epidemiology</subject><subject>Autoantibodies</subject><subject>Autoantibodies - blood</subject><subject>Biomarkers - blood</subject><subject>Biopsy</subject><subject>cardiolipin</subject><subject>Chi-Square Distribution</subject><subject>Cluster Analysis</subject><subject>Connective tissue diseases</subject><subject>Disease Progression</subject><subject>Esophagus</subject><subject>Familial Primary Pulmonary Hypertension</subject><subject>Female</subject><subject>Fibrosis</subject><subject>Fractures</subject><subject>Hospitals</subject><subject>Humans</subject><subject>Hungary - epidemiology</subject><subject>Hypertension</subject><subject>Hypertension, Pulmonary - epidemiology</subject><subject>Immunoglobulin M</subject><subject>Immunology</subject><subject>Incidence</subject><subject>Investigations</subject><subject>Longitudinal Studies</subject><subject>Lung diseases</subject><subject>Lung Diseases, Interstitial - epidemiology</subject><subject>Lupus</subject><subject>Male</subject><subject>Medical prognosis</subject><subject>Middle Aged</subject><subject>Mixed connective tissue disease</subject><subject>Mixed Connective Tissue Disease - classification</subject><subject>Mixed Connective Tissue Disease - diagnosis</subject><subject>Mixed Connective Tissue Disease - epidemiology</subject><subject>Mixed Connective Tissue Disease - immunology</subject><subject>Mixed Connective Tissue Disease - mortality</subject><subject>Myositis</subject><subject>Myositis - epidemiology</subject><subject>Osteoporosis</subject><subject>Phenotype</subject><subject>Prevalence</subject><subject>Prognosis</subject><subject>Pulmonary arteries</subject><subject>Pulmonary artery</subject><subject>Raynaud disease</subject><subject>Raynaud Disease - epidemiology</subject><subject>Rheumatoid factor</subject><subject>Survival</subject><subject>Survival Analysis</subject><subject>Thrombosis</subject><subject>Time Factors</subject><issn>0961-2033</issn><issn>1477-0962</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2012</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>AZQEC</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><recordid>eNqNkU1Lw0AQhhdRbK3ePUnAi5fofm_iTeonFL3oOWyyk7olTWImKfbfm9AqUhA8DTPzzPsyM4ScMnrJmDFXNNaMUyEYl0obxfbImEljwr7O98l4aIdDf0SOEBeUUsFifUhGnEdacqHH5PnWY-vLrA3qdyirdl0DBr4Mlv4TXJBVZQlZ61cQtB6xg8B5BItwHWCXzpuqqzGwpeuzZuVXtjgmB7ktEE62cULe7u9ep4_h7OXhaXozCzOpZRtGltKYqzjORJSD0swpx50UqXAOYpZGNlUgwCjTI0AVt8qkOZVScccVE2JCLja6dVN9dIBtsvSYQVHYEqoOk_46LIqMMfE_UKaoFkIOquc76KLqmrJfZKCk0TqmpqfohsqaCrGBPKkbv7TNOmF0MDbJ7lv6kbOtcJcuwf0MfP-hB8INgHYOv1z_EvwCifuTDw</recordid><startdate>20121101</startdate><enddate>20121101</enddate><creator>Szodoray, P</creator><creator>Hajas, A</creator><creator>Kardos, L</creator><creator>Dezso, B</creator><creator>Soos, G</creator><creator>Zold, E</creator><creator>Vegh, J</creator><creator>Csipo, I</creator><creator>Nakken, B</creator><creator>Zeher, M</creator><creator>Szegedi, G</creator><creator>Bodolay, E</creator><general>SAGE Publications</general><general>Sage Publications Ltd</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7T5</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>H94</scope><scope>K9-</scope><scope>K9.</scope><scope>M0R</scope><scope>M0S</scope><scope>M1P</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope></search><sort><creationdate>20121101</creationdate><title>Distinct phenotypes in mixed connective tissue disease: subgroups and survival</title><author>Szodoray, P ; Hajas, A ; Kardos, L ; Dezso, B ; Soos, G ; Zold, E ; Vegh, J ; Csipo, I ; Nakken, B ; Zeher, M ; Szegedi, G ; Bodolay, E</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c464t-8a0092599c38fe561d5d2d43b3dde91b8ab5e3e75799ce052a57bf04452d25133</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2012</creationdate><topic>Adult</topic><topic>Age</topic><topic>Aged</topic><topic>Analysis of Variance</topic><topic>Angina</topic><topic>Antibodies</topic><topic>Arthritis</topic><topic>Arthritis - epidemiology</topic><topic>Autoantibodies</topic><topic>Autoantibodies - blood</topic><topic>Biomarkers - blood</topic><topic>Biopsy</topic><topic>cardiolipin</topic><topic>Chi-Square Distribution</topic><topic>Cluster Analysis</topic><topic>Connective tissue diseases</topic><topic>Disease Progression</topic><topic>Esophagus</topic><topic>Familial Primary Pulmonary Hypertension</topic><topic>Female</topic><topic>Fibrosis</topic><topic>Fractures</topic><topic>Hospitals</topic><topic>Humans</topic><topic>Hungary - epidemiology</topic><topic>Hypertension</topic><topic>Hypertension, Pulmonary - epidemiology</topic><topic>Immunoglobulin M</topic><topic>Immunology</topic><topic>Incidence</topic><topic>Investigations</topic><topic>Longitudinal Studies</topic><topic>Lung diseases</topic><topic>Lung Diseases, Interstitial - epidemiology</topic><topic>Lupus</topic><topic>Male</topic><topic>Medical prognosis</topic><topic>Middle Aged</topic><topic>Mixed connective tissue disease</topic><topic>Mixed Connective Tissue Disease - classification</topic><topic>Mixed Connective Tissue Disease - diagnosis</topic><topic>Mixed Connective Tissue Disease - epidemiology</topic><topic>Mixed Connective Tissue Disease - immunology</topic><topic>Mixed Connective Tissue Disease - mortality</topic><topic>Myositis</topic><topic>Myositis - epidemiology</topic><topic>Osteoporosis</topic><topic>Phenotype</topic><topic>Prevalence</topic><topic>Prognosis</topic><topic>Pulmonary arteries</topic><topic>Pulmonary artery</topic><topic>Raynaud disease</topic><topic>Raynaud Disease - epidemiology</topic><topic>Rheumatoid factor</topic><topic>Survival</topic><topic>Survival Analysis</topic><topic>Thrombosis</topic><topic>Time Factors</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Szodoray, P</creatorcontrib><creatorcontrib>Hajas, A</creatorcontrib><creatorcontrib>Kardos, L</creatorcontrib><creatorcontrib>Dezso, B</creatorcontrib><creatorcontrib>Soos, G</creatorcontrib><creatorcontrib>Zold, E</creatorcontrib><creatorcontrib>Vegh, J</creatorcontrib><creatorcontrib>Csipo, I</creatorcontrib><creatorcontrib>Nakken, B</creatorcontrib><creatorcontrib>Zeher, M</creatorcontrib><creatorcontrib>Szegedi, G</creatorcontrib><creatorcontrib>Bodolay, E</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Immunology Abstracts</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central Essentials</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>Consumer Health Database (Alumni Edition)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Consumer Health Database</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>MEDLINE - Academic</collection><jtitle>Lupus</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Szodoray, P</au><au>Hajas, A</au><au>Kardos, L</au><au>Dezso, B</au><au>Soos, G</au><au>Zold, E</au><au>Vegh, J</au><au>Csipo, I</au><au>Nakken, B</au><au>Zeher, M</au><au>Szegedi, G</au><au>Bodolay, E</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Distinct phenotypes in mixed connective tissue disease: subgroups and survival</atitle><jtitle>Lupus</jtitle><addtitle>Lupus</addtitle><date>2012-11-01</date><risdate>2012</risdate><volume>21</volume><issue>13</issue><spage>1412</spage><epage>1422</epage><pages>1412-1422</pages><issn>0961-2033</issn><eissn>1477-0962</eissn><abstract><![CDATA[The aim of the present study was to assess the autoantibody profile, dominant clinical symptoms and cluster characteristics of different Mixed connective tissue disease (MCTD phenotypes. Two-hundred-and-one patients with MCTD were followed-up longitudinally. Five clinical parameters, Raynaud’s phenomenon, pulmonary artery hypertension (PAH), myositis, interstitial lung disease (ILD), erosive arthritis and five auto-antibodies besides anti-U1RNP, antiendothelial cell antibodies (AECA), anti-CCP, anti-cardiolipin (anti-CL), anti-SSA/SSB and IgM rheumatoid factor (RF) were selected for cluster analysis. The mean age of patients was 52.9 ± 12.4 years and the mean follow-up of the disease was 12.5 ± 7.2 years. Patients were classified into three cluster groups. Cluster 1 with 77 patients, cluster 2 with 79 patients and cluster 3 with 45 patients. In cluster 1 the prevalence of PAH (55.8%; p < 0.001), Raynaud’s phenomenon (92.2%; p < 0.001) and livedo reticularis (24.6%, p < 0.001) was significantly greater than in cluster 2 and 3. In cluster 2, the incidence of ILD (98.7%; p < 0.001), myositis (77.2%; p < 0.001), and esophageal dysmotility (89.8%; p < 0.001) was significantly greater than that in cluster 1 and 3. In cluster 3, anti-CCP antibodies were present in 31 of 45 patients (68.8%) with erosions. Anti-CCP antibodies were present in 37 of 42 patients (88.0%) with erosions. PAH, angina, venous thrombosis was observed in cluster 1 and pulmonary fibrosis in cluster 2, musculosceletal damage, gastrointestinal symptoms and osteoporotic fractures were most frequent in cluster 3. Cumulative survival assessment indicated cluster 1 patients having the worst prognosis. Cluster analysis is valuable to differentiate among various subsets of MCTD and useful prognostic factor regarding the disease course.]]></abstract><cop>London, England</cop><pub>SAGE Publications</pub><pmid>22864236</pmid><doi>10.1177/0961203312456751</doi><tpages>11</tpages></addata></record> |
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| ispartof | Lupus, 2012-11, Vol.21 (13), p.1412-1422 |
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| source | MEDLINE; SAGE Complete A-Z List |
| subjects | Adult Age Aged Analysis of Variance Angina Antibodies Arthritis Arthritis - epidemiology Autoantibodies Autoantibodies - blood Biomarkers - blood Biopsy cardiolipin Chi-Square Distribution Cluster Analysis Connective tissue diseases Disease Progression Esophagus Familial Primary Pulmonary Hypertension Female Fibrosis Fractures Hospitals Humans Hungary - epidemiology Hypertension Hypertension, Pulmonary - epidemiology Immunoglobulin M Immunology Incidence Investigations Longitudinal Studies Lung diseases Lung Diseases, Interstitial - epidemiology Lupus Male Medical prognosis Middle Aged Mixed connective tissue disease Mixed Connective Tissue Disease - classification Mixed Connective Tissue Disease - diagnosis Mixed Connective Tissue Disease - epidemiology Mixed Connective Tissue Disease - immunology Mixed Connective Tissue Disease - mortality Myositis Myositis - epidemiology Osteoporosis Phenotype Prevalence Prognosis Pulmonary arteries Pulmonary artery Raynaud disease Raynaud Disease - epidemiology Rheumatoid factor Survival Survival Analysis Thrombosis Time Factors |
| title | Distinct phenotypes in mixed connective tissue disease: subgroups and survival |
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